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Glioblastoma multiforme (GBM) is a WHO grade 4 primary brain tumor with a recalcitrant and dismal prognosis and a 14-month-median survival time. The extracranial spread of GBM is so rare that historically it was not believed to spread outside of the central nervous system (CNS). Since the first extracranial spread of GBM was described in 1928, more cases have been reported. However, the mechanisms have yet to be elucidated due to the rareness of well-documented cases. Here, we reported two cases of GBM with postoperative extracranial spread and reviewed related literature.